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A Case of Targetoid Hemosideric Hemangioma Gina Kim, M.D., Hwa-Jung Ryu, M.D., Chil-Hwan Oh, M.D.

Department of Dermatology, College of Medicine, Korea University

Targetoid hemosideric hemangioma represents a distinctive, benign, vascular tumor. His- tologically, it is a noncircumscribed vascular proliferation that may extend into the subcutaneous tissue. The superficial component is composed of ectactic dermal vascular lumina with intra- dermal papillary projections. The deeper component is composed of angular lumina dissecting collagen bundles. We present a case of targetoid hemosideric hemangioma which was presented as solitary nodule on left thigh. (Ann Dermatol 14(2) 124-126, 2002).

Key Words : Targetoid hemosideric hemangioma

Targetoid hemosideric hemangiomas, also known as and eosin staining. Histologic examination re- hobnail hemangioma, are benign vascular tumors vealed ectactic vessels packed with many red characterized histologically by a biphasic growth blood cells in the superficial dermis and some pap- pattern of dilated vascular structures in the superfi- illary projection is identified. There was some lym- cial dermis lined by prominent hobnail endothe- phocytic infiltration and extravasation of erythro- lial cells and collagen dissecting rather narrow cytes is prominent. In upper dermis, endothelial neoplastic vessels in deeper part of the lesion1,2. cells which protruded into lumina resembled hob- Peripheral deposition of hemosiderin gives the lesion nails were seen(Fig. 2A). In deeper dermis, slit- a fairly characteristic targetoid appearance2. shaped thin walled vessels appeared dissecting the cellagen bundle(Fig. 2B). CASE REPORT After biopsy, the eccymotic ring has disappeared over time and she has shown no recurrence until A 54-year-old woman presented to us with a vio- now. laceous papule with halo on left thigh. She said it de- veloped about five days ago suddenly and denied DISCUSSION any history of trauma, irritation or symptoms in this solitary lesion. Her past history and family Targetoid hemosideric hemangioma recently de- history were not remarkable. The routine laborato- scribed benign vascular tumor. It was originally ry evaluation revealed no specific findings. Physical described in 1988 by Santa Cruz and Aronberg3. evaluation revealed a solitary, asymptomatic, After the original description, a few additional match-head sized violaceous papule, surrounded case reports have appeared in the literature and by thin, pale area and a peripheral ecchymotic now it is regarded as a new, unique benign vascular ring on left thigh(Fig. 1). neoplasm. The term ‘targetoid hemosideric he- Under the impression of irritated hemangioma, mangioma’ resulted from characteristic targetoid the central papule was excised completely and ex- appearance on clinical examination1. However, it amined under the microscope with hematoxylin has been shown clearly that most vascular lesions showing histologic features of ‘targetoid hemo- Received July 27, 2001. sideric hemangioma’ lack this clinical appearance. In Accepted for publication October 31, 2002. order to emphasize the diagnostic hobnail cyto- Reprint request to : Gina Kim, M.D., Department of morphology of these neoplasms independent of Dermatology, College of Medicine, Korea University the clinical targetoid appearance, the alternative 80 Guro-Dong, Guro-gu, Seoul, 152-703 name hobnail hemangioma was proposed4,5. Ac- A Case of Targetoid Hemosideric Hemangioma 125

that targetoid hemosideric hemangiomas represent lesions of vascular endothelium2. Factor VIII-asso- ciated antigen has been variously reported as weakly positive and as negative3,4. Several authors postulate that targetoid hemosideric hemangiomas are the result of trauma to preexisting heman- giomas with subsequent occurrence of thrombi and recanalization6,7. Histologically, targetoid hemosideric heman- gioma shows two distinctive features. The first is a hobnail endothelial cytomorphologic appearance, and the second is a biphasic growth pattern. In the superficial dermis, dilated, irregular, thin- Fig. 1. Match-head sized violaceous papule, surround- walled ectactic vascular spaces are observed. The ed by ecchymotic ring on left thigh. vascular spaces sometimes exhibit intraluminal papillary projections and often lined by bland en- cording to some literature, hobnail hemangioma dothelial cells with scanty cytoplasm and round and targetoid hemosideric hemangioma are de- nuclei that protrude into the lumina and closely scribed as a same disease entity5. resemble hobnails. In the deeper dermis, the vascu- Targetoid hemosideric hemangioma presents lar channels become less conspicuous, slit shaped, ir- clinically as a brown to violaceous central papule, regular and angulated, so they appear to dissect surrounded by a thin, pale area and a peripheral between collagen bundles. Extensive extravasa- ecchymotic ring, which is usually less than 1cm in tion of erythrocytes and in a later stage, extensive diameter. It has a characteristic clinical presentation stromal hemosiderin deposition are commonly of the acute onset of a small circular lesion with a seen. A variable degree of lymphocytic infiltration is raised purple center and successive clear and ec- observed. chymotic halos that expand peripherally over Histologic differential diagnosis includes those time. It can arise in any site, but commonly in the hemangiomas that have hobnail endothelial cells extremities and the trunk of young or middle aged such as retiform hemangioendothelioma, malig- adults with slightly male predominance. nant endovascular papillary angioendothelioma, The clinical differential diagnosis included patch stage Kaposi’s sarcoma, epithelioid heman- melanocytic nevus, dermatofibroma, hemangioma, gioma, and progressive lymphangioma2. The pres- and insect bite reaction6. ence of plasma cells, apoptotic endothelial cells, As far as histogenesis is concerned, it is likely and intracytoplasmic endothelial inclusion bodies fa-

(A) (B)

Fig. 2. A) Large vascular spaces with a single layer of endothelial cells and papillary projections(H & E, × 100). B) Slit-like vascular spaces dissecting collagen bundles(H & E, × 200). Annals of Dermatology 126 Gina Kim, et al. Vol. 14, No. 2, April 2002 vor Kaposi’s sarcoma3,8. Most cases of Kaposi’s sar- 62 cases. J Cutan Pathol 1999;26:279-286 coma can be distinguished by the presence of mul- 3. Santa Cruz DJ, Aronberg J. Targetoid hemosideric tiple non-targetoid purplish macules or nodules. hemangioma. J Am Acad Dermatol 1988;19:550- Epithelioid hemangioma may be identified by its 558 significant inflammatory component, with lym- 4. Calonje E, Fletcher CDM, Wilson-Jones E, Rosai J. phocytes and eosinophils in the interstitium and Retiform hemangioendothelioma. A distinctive generally demonstrates few papillary projections1. form of low-grade angiosarcoma delineated in a se- In our case, no inflammatory component was ob- ries of 15 cases. Am J Surg Pathol 1994;18:115-125 served. Progressive lymphangioma may be con- 5. Guillou L, Calonje E, Speight P, Rosai J, Fletcher fused with targetoid hemosideric hemangioma be- CDM. Hobnail hemangioma: a pseudomalignant cause it also demonstrates slit shaped, angulated vascular lesion with a reappraisal of targetoid he- lumina. This lesion, however, lacks the hemo- mosideric hemangioma. Am J Surg Pathol 1999; siderin deposition and the superficial “hobnailed” en- 23:97-105 dothelial cells9,10. Our case shows hemosiderin de- 6. Rapini RP, Golitz LE. Targetoid hemosideric he- posit and hobnailed endothelial cells. Clinically, mangioma. J Cutan Pathol 1990;17:233-235 it is an erythematous patch or plaque, much larger in 7. Requena L, Sanguenza OP. Cutaneous vascular size than targetoid hemosideric hemangioma. proliferation. Part II. Hyperplasias and benign neo- In conclusion, we present a typical form of plasms. J Am Acad Dermatol 1997;37:887-919 targetoid hemosideric hemangioma. 8. Chor PJ, Santa Cruz DJ. Kaposi’s sarcoma: a clini- copathologic review and differential diagnosis. J REFERENCE Catan Pathol 1992;19:6-20 9. Watanabe M, Kishiyama K, Ohkawara A. Acquired 1. Vion B, Frenk E. Targetoid hemosideric heman- progressive lymphangioma. J Am Acad Dermatol gioma. Dermatology 1992; 184:300-302 1983;80:663- 2. Thomas M, Taina AP, Heinz K. Hobnail heman- 10. Wilson-Jones E, Winkelmann RK, Zachary CB, Re- gioma(“targetoid hemosideric hemangioma”):clini- da AM. Benign lymphangioendothelioma J Am copathologic and immunohistochemical analysis of Acad Dermatol 1990;23:229-